Case Report EctopicHidradenomaPapilliferum

Hindawi Publishing CorporationDermatology Research and PracticeVolume 2010, Article ID 709371, 3 pagesdoi:10.1155/2010/709371

Case Report

Ectopic Hidradenoma Papilliferum

Aristoteles David Neiva Rosmaninho,1 Maria Teresa Duarte Pinto de Almeida,1

Vırgilio Costa,1 Maria Madalena Vasconcelos Sanches,1

Carlos Lopes,2 and Maria Manuela Selores Gomes Meirinhos1

1 Departamento de Dermatologia, Centro Hospitalar do Porto, HSA, Edifıcio das Consultas Externas, Ex-CICAP, Rua D. Manuel II,4099-001 Porto, Portugal

2 Departamento de Anatomia Patologica, Centro Hospitalar do Porto, HSA, Porto, 4099-001 Porto, Portugal

Correspondence should be addressed to Aristoteles David Neiva Rosmaninho, [email protected]

Received 23 August 2010; Revised 2 November 2010; Accepted 17 November 2010

Academic Editor: K. Yamanishi

Copyright © 2010 Aristoteles David Neiva Rosmaninho et al. This is an open access article distributed under the CreativeCommons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided theoriginal work is properly cited.

Hidradenoma papilliferum is a rare tumor that occurs almost exclusively in females on the anogenital area. Rare cases of ectopic(nongenital) hidradenoma papilliferum have been described. The lesions usually present as an asymptomatic slow-growing,red, firm, mobile, well-delimitated nodule that grows for a long time before resection. We describe a case of an 26-year-oldman that presented with an enlarging nodule on his right eyelid. The histological findings revealed a hidradenoma papillif-erum. So far, among the very few reports of ectopic hidradenoma papilliferum, only a very small number were localized to theeyelid.

1. Introduction

Hidradenoma papilliferum is a slow-growing benign adnexaltumor with apocrine differentiation, and for some authors itcan be considered an analog of intraductal papilloma of thebreast [1]. The tumor primarily affects vulvar, perineal andperianal skin of middle-aged women with rare cases beingreported in other skin localizations (ectopic hidradenomapapilliferum) [2, 3]. To our knowledge, only a perianal,hidradenoma papilliferum has been described in a man [4].The clinical presentation as well as the pathologic features,treatment, and prognosis are similar in both forms. Itusually presents as a slow-growing, solitary, asymptomaticskin colored or red nodule less than 1 cm in diameter [5].However, a giant ectopic hidradenoma papilliferum on thescalp has been recently described [6]. The most commonsite of ectopic hidradenoma papilliferum is the head andneck. The diagnosis can be made only by histopathologicalexamination since they clinically mimic other cutaneousneoplasms.

2. Clinical History

A 26-year-old white man presented in our departmentwith a 2-year history of an asymptomatic enlarging lesionof the upper eyelid. No previous lesion, namely nevussebaceous was documented. His personal and familiarmedical history was unremarkable. On physical examinationa well-circumscribe, firm, umbilicated, reddish, molluscumcontagiosum-like nodule approximately 1 cm in diameterlocalized to the upper right eyelid was observed (Figure 1).Dermoscopy showed several telangiectasias in the surface ofthe nodule and a central umbilication (Figure 2). A basal cellcarcinoma or an adnexal skin tumor was suspected. Surgicalexcision of the lesion was performed and the specimen wassent for microscopic examination.

Microscopic examination showed a well-circumscribeddermal nodule characterized by a large cystic space con-taining eosinophilic debris and papillary folds. The lesionseemed to be connected with the epidermis and a cen-tral ulceration was observed. Papillae and micropapillae

2 Dermatology Research and Practice

Figure 1: Clinical features of the skin lesion on the upper eyelid.

Figure 2: Dermoscopy findings.

Figure 3: Hematoxylin-eosin stain (4x) showed a dermal nodulewith many glandular structures and papillary folds.

Figure 4: Hematoxylin-eosin stain (40x) showed the presence offocal areas infiltrated by plasma cells and lymphocytes in thestroma.

Figure 5: Hematoxylin-eosin stain (20x) showed the tumor epithe-lium to be composed by an outer cuboidal cell layer and an innercolumnar cell layer.

projected from the cyst wall into its cavity (Figure 3). Thepapillae had a broad fibrous core with aggregates of lympho-cytes and few plasma cells (Figure 4). A basal layer composedby small cuboidal cells and a luminal layer composedby larger columnar cells was observed (Figure 5). Thesemicroscopic features were consistent with the diagnosis ofhidradenoma papilliferum.

3. Discussion

Hidradenoma papilliferum is originated in the apocrineglands, which are mainly concentrated in the anogenitalregion, axillae, and periumbilical areas. The tumor occursmainly in those areas with ectopic localization being rarelyreported [2]. The distribution of the ectopic forms corre-sponds to the areas containing heterotrophic and modifiedapocrine glands. According to a medline search, only 20reports of ectopic hidradenoma papilliferum have been

Dermatology Research and Practice 3

described in the English language and only 3 were localizedto the eyelid [7–9]. Independently of being typical or ectopicthe tumor occurs mostly in white women. However, in con-trast to anogenital hidradenoma papilliferum, nearly one-half of the patients with ectopic hidradenoma papilliferumare men [2]. The head and neck are the most frequentlocalization for the ectopic presentation, mainly the eyelidand external ear, where modified apocrine glands (Moll andceruminous glands) are found normally [2, 7]. Sometimesectopic apocrine tumors are also found in the scalp withinlesions such as the nevus sebaceous of Jadassohn. There are4 reported cases of hidradenoma papilliferum on the headand neck region in males [7]. Other ectopic localizationsincluded arm, thigh, back, and nipple [2, 7, 10]. The agerange reported is between 8 to 78 years [7]. The clinicalpresentation of both forms is similar to most lesions, beingasymptomatic and growing for a long time before excision.Pain, pruritus or, ulceration can occur. Like other adnexalskin tumors they clinically mimic other neoplasms such asbasal cell carcinoma (as in our case report) and spinocellularcarcinoma. Thus, histological examination is required for thecorrect diagnosis. Histologically the tumor is characterizedby a cystic space containing eosinophilic material and pap-illary folds projected from the cyst wall. Tumor epitheliumis composed by a basal layer of cuboidal cells and a luminallayer of larger columnar cells showing decapitation secretion[11]. The epidermis may be normal, acanthotic, or ulceratedand may sometimes show continuity with the overlyingepithelium [12, 13]. In some cases the tumor displays ahistopathology similar to syringocystadenoma papilliferumsince they are closely related tumors which originate fromapocrine glands [14]. Aggregates of lymphocytes and plasmacells have been described in the stroma of ectopic lesions[2]. Some authors considered the presence of focal areasinfiltrated by plasma cells and lymphocytes as a sign ofa mixed differentiation between hidradenoma papilliferumand syringocystadenoma papilliferum [7]. A report of anectopic hidradenoma papilliferum with sebaceous differen-tiation has been documented [8]. Other histopathologicaldifferential diagnosis includes tubular apocrine adenomaand clear cell (apocrine) adenoma. The prognosis is goodwith local excision being the treatment of choice. Recur-rence of the lesions is attributed to incomplete excision ofthe primary tumor and there is no report of recurrencefor the ectopic form [2]. Malignant transformation inanogenital hidradenoma papilliferum has been documented(intraductal carcinoma resembling apocrine carcinoma andinvasive adenosquamous carcinoma) but not in the ectopicpresentation [2, 15, 16]. It is speculated that HPV mayplay a role in inducing malignancy, but the associationstill needs to be proved [17]. We presented a new case ofectopic hidradenoma papilliferum with features of a mixeddifferentiation arising in the eyelid.

References

[1] S. C. J. van der Putte, “Mammary-like glands of the vulvaand their disorders,” International Journal of GynecologicalPathology, vol. 13, no. 2, pp. 150–160, 1994.

[2] R. Vang and P. R. Cohen, “Ectopic hidradenoma papilliferum:a case report and review of the literature,” Journal of theAmerican Academy of Dermatology, vol. 41, no. 1, pp. 115–118,1999.

[3] D. J. Santa Cruz, P. G. Prioleau, and M. E. Smith, “Hidrade-noma papilliferum of the eyelid,” Archives of Dermatology, vol.117, no. 1, pp. 55–56, 1981.

[4] J. Loane, W. F. Kealy, and G. Mulcahy, “Perianal hidradenomapapilliferum occurring in a male: a case report,” Irish Journalof Medical Science, vol. 167, no. 1, pp. 26–27, 1998.

[5] J. H. Meeker, R. D. Neubecker, and E. B. Helwig, “Hidrade-noma papilliferum,” American Journal of Clinical Pathology,vol. 37, pp. 182–195, 1962.

[6] J. W. Moon, C. H. Na, H. R. Kim, and B. S. Shin, “Giantectopic hidradenoma papilliferum on the scalp,” Journal ofDermatology, vol. 36, no. 10, pp. 545–547, 2009.

[7] M. J. Fernandez-Acenero, T. Aramendi Sanchez, M. C.Villanueva Sanchez, and L. Requena, “Ectopic hidradenomapapilliferum: a case report and literature review,” AmericanJournal of Dermatopathology, vol. 25, no. 2, pp. 176–178, 2003.

[8] S. Minami, N. Sadanobu, T. Ito, M. Natsuaki, and K. Yaman-ishi, “Non-anogenital (ectopic) hidradenoma papilliferumwith sebaceous differentiation: a case report and review ofreported cases,” Journal of Dermatology, vol. 33, no. 4, pp. 256–259, 2006.

[9] E. J. Lee, M. K. Shin, C. R. Haw, and M. H. Lee, “Two casesof hidradenoma papilliferum of the nose,” Acta Dermato-Venereologica, vol. 90, no. 3, pp. 322–323, 2010.

[10] M. Tanaka and S. Shimizu, “Hidradenoma papilliferumoccurring on the chest of a man,” Journal of the AmericanAcademy of Dermatology, vol. 48, no. 2, supplement, pp. S20–S21, 2003.

[11] E. J. Wilkinson and D. L. Xie, “Benign diseases of the vulva,” inBlaustein S Pathology of the Female Genital Tract, R. J. Kurman,Ed., pp. 37–98, Springer, New York, NY, USA, 5th edition,2002.

[12] A. Virgili, A. Marzola, and M. Corazza, “Vulvar hidradenomapapilliferum: a review of 10.5 years’ experience,” Journal ofReproductive Medicine for the Obstetrician and Gynecologist,vol. 45, no. 8, pp. 616–618, 2000.

[13] R. L. Warkel, “Selected apocrine neoplasms,” Journal ofCutaneous Pathology, vol. 11, no. 5, pp. 437–449, 1984.

[14] W. Nishie, D. Sawamura, M. Mayuzumi, S. Takahashi,and H. Shimizu, “Hidradenoma papilliferum with mixedhistopathologic features of syringocystadenoma papilliferumand anogenital mammary-like glands,” Journal of CutaneousPathology, vol. 31, no. 8, pp. 561–564, 2004.

[15] P. Bannatyne, P. Elliott, and P. Russell, “Vulvar adenosqua-mous carcinoma arising in a hidradenoma papilliferum, withrapidly fatal outcome: case report,” Gynecologic Oncology, vol.35, no. 3, pp. 395–398, 1989.

[16] G. Pelosi, G. Martignoni, and F. Bonetti, “Intraductal carci-noma of mammary-type apocrine epithelium arising withina papillary hydradenoma of the vulva: report of a case andreview of the literature,” Archives of Pathology and LaboratoryMedicine, vol. 115, no. 12, pp. 1249–1254, 1991.

[17] M. Vazmitel, D. V. Spagnolo, J. Nemcova, M. Michal, and D. V.Kazakov, “Hidradenoma papilliferum with a ductal carcinomain situ component: case report and review of the literature,”American Journal of Dermatopathology, vol. 30, no. 4, pp. 392–394, 2008.

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